Multifocal Cystic Bone Tuberculosis with Lupus Vulgaris and Lymphadenitis

INDIAN PEDIATRICS VOLUME 34-MAY 1997

via cavernous sinus and intracranial propa- CT or an MRI should be done as the inves- gation of periorbital cellulitis. This was not tigation of choice to differentiate between required in our case due to probably insti- an inflammatory lesion and a tumor. tution of appropriate antibiotic therapy. It may be of merit in severe or late diagnosed REFERENCES cases. Staphylococcal colonization of na- 1. Besley G, Minns RA. Disorders of the cen- sopharynx can lead to ethmoiditis in tral nervous system. In: Forfar and immunocompromized or susceptible child, Ameil's Textbook of Pediatrics, 4th edn. with rapid progress locally and hematoge- Eds. Brown JK, Campbell AGM, Mclntosh nous seeding as in our case. A variety of or- N. Edinburgh, Churchill Livingstone, ganisms other than staphylococcus can 1992, pp 898-901. cause this infection, which is dangerous be- 2. Haynes RE, Cramblett HG. Acute cause it may be complicated by retrobulbar ethmoiditis: Its relationship to orbital cellulitis. Am J Dis Child 1967; 114: 261- abscess and cavernous sinus infection and 267. thrombosis. Treatment should be started 3. Moranne JE, Estorunet B, Adrien A, early with intravenous antibiotics. Also, Seurat MC, Barois A. Staphylococcus, the concomitant staphylococcal foci should be most frequent agent of serious complica- looked for and dealt with surgically if nec- tions of acute sinusitis in children: 5 cases. essary. If in any doubt, a contrast enhanced Ann Intern Med 1982; 133: 462-467.

Multifocal Cystic Bone Tuberculosis Rarer still is its association with skin tuber- with Lupus Vulgaris and culosis and lymphadenopathy. Here we are Lymphadenitis reporting a case of multiple cystic bone tuberculosis with lupus vulgaris and lymphadenopathy. Anju Aggarwal Case Report S. Aneja A 2V2-year-old male child presented V. Taluja with low grade fever and painless swelling Rama Anand* of hands and feet for ten months. Three brownish raised lesions on skin and multiple swellings of groin and neck were Multifocal cystic bone tuberculosis in- present for the last 6 months. He had been volving axial and extra-axial skeleton is a immunized for BCG and had a history of rare form of this disease entity in children. contact with a patient of tuberculosis in the From the Departments of Pediatrics and Radiology*, neighborhood. Examination revealed a Kalawati Saran Children's Hospital and Lady child of average built and nutrition, mild Hardinge Medical College, New Delhi 110 001. pallor, 3-4 firm, nontender and mobile pal- Reprint requests: Dr. Anju Aggarwal, Flat No. 3C, pable lymphnodes 1-2 cm in size in right Block C2B, Janakpuri, New Delhi 110 058. cervical region, groin and left axilla. Nodu- Manuscript Received: October 1,1996; lar, brownish lesion 2 x 6 cm was seen on Initial review completed: November 28,1996; the right buttock and similar lesions were Revision Accepted: December 11,1996 documented on left toe and right leg. The

443 CASE REPORTS region of hand over metacarpals and meta- scales over skin lesions cleared within two tarsals was swollen. Spine examination re- weeks of treatment. Cystic lesions of tibia vealed a depression in the region of T7-T8 and fibula showed radiological improve- vertebrae. Examination of abdomen, car- ment after four months. The child gained diovascular and respiratory systems did height and weight normally as observed on not reveal any abnormality. follow up. On investigation, child was found to Discussion have a hemoglobin of 8.5 g/dl, ESR of 65 mm, Mantoux of 12 x 12 mm. Chest X-ray With the advent of effective chemother- revealed a fibrotic band in the right upper apy for tuberculosis, the incidence of bone zone with areas of spotty calcification in tuberculosis has declined. The most fre- right hilar region, destruction of D8 verte- quent site of involvement in skeletal tuber- brae with bilateral paravertebral abscesses. culosis is spine followed by joints. Tubercu- Cystic lesions were seen in metacarpals of lous dactylitis and cystic tuberculosis are both hands, shafts of tibia and fibula, meta- rare(l). They represent special forms of tu- tarsals and cuboid bone (Figs. 1 & 2). X-ray berculous osteomyelitis. Various terminol- skull did not reveal any abnormality. Fine ogies have been used for multiple cystic tu- needle aspiration cytology (FNAC) from berculosis including Jungling's disease, cervical lymphnode revealed caseating pseudocystic tuberculosis in children and granuloma with acid fast bacilli. Skin biop- disseminated bone disease in adults(2). In sy was suggestive of lupus vulgaris. ELISA children, multiple focal lesions are more common and peripheral skeleton is more for HIV was negative. commonly involved than axial skeleton as The child was treated with four compared to adults. Lesions in shaft or antitubercular drugs (streptomycin, pyra- metaphysis of long bones as seen in our zinamide, isoniazid and rifampicin) for two case are seen in less than 1% cases(l). The months and later isoniazid and rifampicin exact incidence of multifocal cystic tuber- was continued. Swellings subsided and culosis is not known but a study has given

Fig. 1. Cystic lesion in first metatarsal.

444 INDIAN PEDIATRICS VOLUME 34-MAY 1997

Fig. 2. Multiple cystic lesions in lower end of tibia, fibula and cuboid bone. an incidence of multifocal osteoarticular tu- of cystic tuberculosis involving only the berculosis in the Indian population to be 7- skull(6,7) with few reports similar to our 10% of skeletal tuberculosis(3). This study case(8). The incidence of skin tuberculosis included children as well as adults with with cystic bone tuberculosis in children is bone involvement, joint involvement or not known. There is one case report of both. Tuberculous dactylitis is more com- lupus vulgaris with multiple bone lesions mon than cystic tuberculosis and its report- due to BCG in 1954(9). It was estimated ed incidence is 0.5 to 14% and is usually that 40% cases of lupus have associated seen in children less than 5 years of age(4). cervical adenitis or lupus of mucous mem- Mantoux positivity varies from 60-80% branes and 10-20% have pulmonary or and most cases have evidence of pulmo- bone involvement(10). Multiple lesions in nary involvement(3). Bone lesions present bone can also be seen due to hematogenous in a bizzare fashion and mimic other diag- spread of bacilli 6-12 months following nosis like pyogenic osteomyelitis, sarcoi- BCG vaccination. They are usually seen in dosis and fungal infection; hence biopsy is children 5 months to 6 years of age. Tubu- usually required to confirm the diagno- lar bones and ribs on side of vaccination sis^). Biopsy was not done in our case as are usually involved. The computed inci- we had evidence of tuberculosis in the dence is 1 in 5000 to 1 in 8000 and these le- form of Mantoux positivity and primary in- sions have a good prognosis. Such lesions volvement of lungs. Skin biopsy and FNAC are not associated with immunological dis- of lymph nodes was diagnostic of Mycobac- orders and diagnosis can be established terium tuberculosis infection. Multiple cystic only by culture of organisms(ll,12). In our lesions usually respond well to chemother- case, pulmonary involvement was seen apy and curettage, and bone grafting is and lesions were not predominant on the rarely required(3). Overall the prognosis of side of vaccination and hence BCG this form of tuberculosis is good. osteomyelitis was not considered as the eti- Most Indian studies have reported cases ology.

445 CASE REPORTS

Both lupus vulgaris and multifocal bone 6. Bhandari B, Mandowara SL, Joshi H. Tu- tuberculosis are seen in patients with good berculous osteomyelitis of skull. Indian J immunity. Response to therapy is excellent Pediatr 1981; 48:113-115. and prognosis is good as seen in our 7. Gupta PK, Sastry VR, Chandramauli BA, case. Tuberculous osteomyelitis of skull. Indian J Pediatr 1981; 48:113-115. REFERENCES 8. Kher AS, Bharucha BA, Kamat JR, Kurkure P. Cystic tuberculosis of bones. 1. Resnick D, Niwayana G. Osteomyelitis, Indian Pediatr 1993; 30: 676-678. septic arthritis and soft tissue infection. Organisms. In: Diagnosis of Bone and 9. Imerslund O, Jonsen T. Lupus vulgaris Joint Disorders, Vol. IV, 3rd edn. Ed. and multiple bone lesions caused by Resnick D. Philadelphia, W.B. Saunders BCG. Acta Tuberculosa Scand 1954; 30: 116-123. Co, 1995, pp 2448-2558. 2. Rasool MN, Govender S, Naidoo KS. Cys- 10. Horwitz O, Christensen S. Numerical esti- tic tuberculosis of bone in children. J Bone mates of extent of lesion in lupus vulgaris Joint Surg (Br) 1994; 76B: 113-117. cutis and their significance for epidemio- logical and clinical research. Am Rev 3. Kumar K, Saxena MB. Multifocal Repsir Dis 1960; 82: 862-867. osteoarticular tuberculosis. Int Orthop 1988; 12:135-138. 11. Bregdahl S, Fellander M, Roberston B. BCG osteomyelitis. Experience in the 4. Hardy JB, Hartmann JR. Tuberculous Stockholm region over the years 1961- dactylitis in childhood prognosis. J 1974. J Bone Joint Surg (Br) 1976; 58: 212- Pediatr 1947; 30:146-149. 216. 5. Versveld GA, Solomon A. A diagnostic 12. Peltola H, Salmi I, Vahvanen V, et al. BCG approach to tuberculosis of bone and vaccination as a cause of osteomyelitis joints. J Bone Joint Surg (Br) 1982; 64B: and subcutaneous abscess. Arch Dis 446-449. Child 1984; 59:157-161.

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